Correspondence
prior trauma might be a predisposing factor in the occurrence of plantar MC. Our case had neither HIV infection nor trauma history. Theoretically, MC could emerge in any skin or mucosal area. It seems that dermatologists are confronted with other atypical locations in daily practice. İlker Aydoğan, MD Oğuz Küçükçakır, MD Aysße Kavak, MD Ümran Yıldırım, MD Departments of Dermatology and Pathology Duzce Medical School Duzce University Duzce Turkey Aysße Kavak, MD Bakırk€ oy Dr Sadi Konuk Research & Training Hospital Department of Dermatology _ 34100 Istanbul, Turkey E-mail: [email protected] Funding source: None. Conflict of interest: None. References 1 Hanson D, Diven DG. Molluscum contagiosum. Dermatol Online J 2003; 9: 2. 2 Fornatora ML, Reich RF, Gray RG, et al. Intraoral molluscum contagiosum: a report of a case and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2001; 92: 318–320. 3 Hindaal CH, van Bijsterveld OP. Molluscum contagiosum of the palpebral conjunctiva. Report of a case. Ophthalmologica 1979; 178: 137–141. 4 Ha SJ, Park YM, Cho SH, et al. Solitary giant molluscum contagiosum of the sole. Pediatr Dermatol 1998; 15: 222– 224. 5 Cohen PR, Tschen JA. Plantar molluscum contagiosum: a case report of molluscum contagiosum occurring on the sole of the foot and a review of the world literature. Cutis 2012; 90: 35–41.
Isolated benign primary cutaneous plasmacytosis
Cutaneous plasmacytosis is a skin disorder characterized by multiple reddish-brown nodules of polyclonal plasma cell proliferation, mainly affecting the trunk in adults. Extracutaneous involvement of organs such as lymph nodes, lungs, and liver is common and is termed systemic plasmacytosis.1 Isolated, localized forms of cutaneous plasmacytosis are extremely rare. ª 2014 The International Society of Dermatology
A 16-year-old Japanese girl presented with a 1-year history of asymptomatic nodules on the dorsum of the hand. No history of significant past medical conditions or previous trauma in the region of the affected lesions was noted. Physical examination revealed reddish nodules 2–5 mm in diameter on the dorsum of the left thumb (Fig. 1a). Laboratory tests revealed no abnormal findings except for mild anemia and a high titer of antinuclear antibody (homogeneous, 92560), with no clinical manifestations suggestive of collagen diseases. Analyses for serum and urine protein indicated no polyclonal or monoclonal gammopathies. Serum interleukin-6 level was normal. Histopathologically, a dense cellular infiltrate was evident in the dermis, comprising largely mature plasma cells (Fig. 1b,c). Immunohistochemical examination revealed that these cells were positive for CD138 (Fig. 1d), and the possibility of monoclonal proliferation of plasma cells was excluded by doublepositive staining for immunoglobulin kappa and lambda light chains (not shown). Skin lesions were entirely excised. Unexpectedly, the titer of antinuclear antibody decreased to 9160 within three months postoperatively. No recurrence has been noted during the 6-month follow-up period. Localized polyclonal plasma cell proliferation in the skin was originally reported in a 7-year-old child in 2002.2 Subsequently, three additional cases have been reported under the name of isolated benign primary cutaneous plasmacytosis in children.3,4 All these cases, including the present, were clinically characterized by isolated reddish nodules or plaques in children (Table 1), and neither hypergammaglobulinemia nor extracutaneous involvement has been noticed in any cases. The clinical characteristics apparently differ from those in typical cutaneous/systemic plasmacytosis, which usually affects adult patients and presents as multiple brown nodules on the trunk. Isolated benign primary cutaneous plasmacytosis in children can thus be considered as a new subtype of cutaneous plasmacytosis and does not appear to carry a risk of progression to systemic involvement, although much longer-term follow-up may be needed. The pathogenesis of isolated benign primary cutaneous plasmacytosis and the key differences from cutaneous/systemic plasmacytosis are unknown. Local triggering factors, such as trauma and insect bites, have not been noted in any previous cases. A peculiar finding in the present case was the elevated level of antinuclear antibody, which decreased after the excision of skin lesions. We are currently unable to explain the relationship between antinuclear antibody production and cutaneous plasma cell proliferation. Plasma cells in the skin may be autoreactive cells involved in antibody generation, although serum levels of antibody might be barely influenced by such small, localized lesions comprising plasma cells. An additional study with accumulation of additional cases is needed to International Journal of Dermatology 2014, 53, e389–e409
e397
e398
Correspondence
(a)
(b)
(c)
(d)
Figure 1 Clinical and histopathological features. (a) Red nodules of varying sizes aggregated on the extensor surface of the left thumb. (b,c) Dense cellular infiltrates comprising mature plasma cells throughout the entire dermis. Hematoxylin and eosin, original magnification: (b) 940, (c) 9400. (d) Infiltrative cells were positive for CD138 (diaminobenzidine, original magnification, 9400) Table 1 Clinical profiles of reported cases
Case
Sex/age
Locations
Disease duration (years)
1 2 3 4 5
F/7 F/15 M/7 F/7 F/16
Axillary and scapular regions Left anterior tibia Right anterior tibia Right buttock Left hand
4 12 2 5 1
Funding sources: None. Conflicts of interest: None. References
F, female; M, male.
verify the possibility of cutaneous plasma cells contributing to autoantibody production. Yohei Shiba, MD Takahiro Satoh, MD, PhD Department of Dermatology National Defense Medical College Tokorozawa Japan Yohei Shiba, MD Department of Dermatology National Defense Medical College, 3-2 Namiki Tokorozawa 359-8513 Japan E-mail: [email protected]
International Journal of Dermatology 2014, 53, e389–e409
1 Leonard AL, Meehan SA, Ramsey D, et al. Cutaneous and systemic plasmacytosis. J Am Acad Dermatol 2007; 56: S38–S40. 2 Arico M, Bongiorno MR. Primary cutaneous plasmacytosis in a child. Is this a new entity? J Eur Acad Dermatol Venereol 2002; 16: 164–167. 3 Gilliam AC, Mullen RH, Oviedo G, et al. Isolated benign primary cutaneous plasmacytosis in children: two illustrative cases. Arch Dermatol 2009; 145: 299–302. 4 Ahn JJ, Yang YS, Shin MK, et al. Case of isolated benign primary cutaneous plasmacytosis in a child. J Dermatol 2011; 38: 364–367.
An unusual case of nevus of Ota combined with nevus spilus
Editor, Nevus of Ota is a dermal melanocytic disorder of unknown cause. Nevus spilus presents as a light tan patch with darkly pigmented, nevomelanocytic macular and/or papular elements. We describe a patient in whom both types of lesion overlapped in the same area.
ª 2014 The International Society of Dermatology
prior trauma might be a predisposing factor in the occurrence of plantar MC. Our case had neither HIV infection nor trauma history. Theoretically, MC could emerge in any skin or mucosal area. It seems that dermatologists are confronted with other atypical locations in daily practice. İlker Aydoğan, MD Oğuz Küçükçakır, MD Aysße Kavak, MD Ümran Yıldırım, MD Departments of Dermatology and Pathology Duzce Medical School Duzce University Duzce Turkey Aysße Kavak, MD Bakırk€ oy Dr Sadi Konuk Research & Training Hospital Department of Dermatology _ 34100 Istanbul, Turkey E-mail: [email protected] Funding source: None. Conflict of interest: None. References 1 Hanson D, Diven DG. Molluscum contagiosum. Dermatol Online J 2003; 9: 2. 2 Fornatora ML, Reich RF, Gray RG, et al. Intraoral molluscum contagiosum: a report of a case and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2001; 92: 318–320. 3 Hindaal CH, van Bijsterveld OP. Molluscum contagiosum of the palpebral conjunctiva. Report of a case. Ophthalmologica 1979; 178: 137–141. 4 Ha SJ, Park YM, Cho SH, et al. Solitary giant molluscum contagiosum of the sole. Pediatr Dermatol 1998; 15: 222– 224. 5 Cohen PR, Tschen JA. Plantar molluscum contagiosum: a case report of molluscum contagiosum occurring on the sole of the foot and a review of the world literature. Cutis 2012; 90: 35–41.
Isolated benign primary cutaneous plasmacytosis
Cutaneous plasmacytosis is a skin disorder characterized by multiple reddish-brown nodules of polyclonal plasma cell proliferation, mainly affecting the trunk in adults. Extracutaneous involvement of organs such as lymph nodes, lungs, and liver is common and is termed systemic plasmacytosis.1 Isolated, localized forms of cutaneous plasmacytosis are extremely rare. ª 2014 The International Society of Dermatology
A 16-year-old Japanese girl presented with a 1-year history of asymptomatic nodules on the dorsum of the hand. No history of significant past medical conditions or previous trauma in the region of the affected lesions was noted. Physical examination revealed reddish nodules 2–5 mm in diameter on the dorsum of the left thumb (Fig. 1a). Laboratory tests revealed no abnormal findings except for mild anemia and a high titer of antinuclear antibody (homogeneous, 92560), with no clinical manifestations suggestive of collagen diseases. Analyses for serum and urine protein indicated no polyclonal or monoclonal gammopathies. Serum interleukin-6 level was normal. Histopathologically, a dense cellular infiltrate was evident in the dermis, comprising largely mature plasma cells (Fig. 1b,c). Immunohistochemical examination revealed that these cells were positive for CD138 (Fig. 1d), and the possibility of monoclonal proliferation of plasma cells was excluded by doublepositive staining for immunoglobulin kappa and lambda light chains (not shown). Skin lesions were entirely excised. Unexpectedly, the titer of antinuclear antibody decreased to 9160 within three months postoperatively. No recurrence has been noted during the 6-month follow-up period. Localized polyclonal plasma cell proliferation in the skin was originally reported in a 7-year-old child in 2002.2 Subsequently, three additional cases have been reported under the name of isolated benign primary cutaneous plasmacytosis in children.3,4 All these cases, including the present, were clinically characterized by isolated reddish nodules or plaques in children (Table 1), and neither hypergammaglobulinemia nor extracutaneous involvement has been noticed in any cases. The clinical characteristics apparently differ from those in typical cutaneous/systemic plasmacytosis, which usually affects adult patients and presents as multiple brown nodules on the trunk. Isolated benign primary cutaneous plasmacytosis in children can thus be considered as a new subtype of cutaneous plasmacytosis and does not appear to carry a risk of progression to systemic involvement, although much longer-term follow-up may be needed. The pathogenesis of isolated benign primary cutaneous plasmacytosis and the key differences from cutaneous/systemic plasmacytosis are unknown. Local triggering factors, such as trauma and insect bites, have not been noted in any previous cases. A peculiar finding in the present case was the elevated level of antinuclear antibody, which decreased after the excision of skin lesions. We are currently unable to explain the relationship between antinuclear antibody production and cutaneous plasma cell proliferation. Plasma cells in the skin may be autoreactive cells involved in antibody generation, although serum levels of antibody might be barely influenced by such small, localized lesions comprising plasma cells. An additional study with accumulation of additional cases is needed to International Journal of Dermatology 2014, 53, e389–e409
e397
e398
Correspondence
(a)
(b)
(c)
(d)
Figure 1 Clinical and histopathological features. (a) Red nodules of varying sizes aggregated on the extensor surface of the left thumb. (b,c) Dense cellular infiltrates comprising mature plasma cells throughout the entire dermis. Hematoxylin and eosin, original magnification: (b) 940, (c) 9400. (d) Infiltrative cells were positive for CD138 (diaminobenzidine, original magnification, 9400) Table 1 Clinical profiles of reported cases
Case
Sex/age
Locations
Disease duration (years)
1 2 3 4 5
F/7 F/15 M/7 F/7 F/16
Axillary and scapular regions Left anterior tibia Right anterior tibia Right buttock Left hand
4 12 2 5 1
Funding sources: None. Conflicts of interest: None. References
F, female; M, male.
verify the possibility of cutaneous plasma cells contributing to autoantibody production. Yohei Shiba, MD Takahiro Satoh, MD, PhD Department of Dermatology National Defense Medical College Tokorozawa Japan Yohei Shiba, MD Department of Dermatology National Defense Medical College, 3-2 Namiki Tokorozawa 359-8513 Japan E-mail: [email protected]
International Journal of Dermatology 2014, 53, e389–e409
1 Leonard AL, Meehan SA, Ramsey D, et al. Cutaneous and systemic plasmacytosis. J Am Acad Dermatol 2007; 56: S38–S40. 2 Arico M, Bongiorno MR. Primary cutaneous plasmacytosis in a child. Is this a new entity? J Eur Acad Dermatol Venereol 2002; 16: 164–167. 3 Gilliam AC, Mullen RH, Oviedo G, et al. Isolated benign primary cutaneous plasmacytosis in children: two illustrative cases. Arch Dermatol 2009; 145: 299–302. 4 Ahn JJ, Yang YS, Shin MK, et al. Case of isolated benign primary cutaneous plasmacytosis in a child. J Dermatol 2011; 38: 364–367.
An unusual case of nevus of Ota combined with nevus spilus
Editor, Nevus of Ota is a dermal melanocytic disorder of unknown cause. Nevus spilus presents as a light tan patch with darkly pigmented, nevomelanocytic macular and/or papular elements. We describe a patient in whom both types of lesion overlapped in the same area.
ª 2014 The International Society of Dermatology
